Parotid carcinoma following chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids: a case report.
Parotid carcinoma following chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids: a case report.
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DOI：
10.1186/s12883-021-02135-6
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发表时间：
2021-03-10
期刊：
影响因子：
2.6
通讯作者：
Liu M
中科院分区：
文献类型：
--
作者：
Deng Y;Zhao B;Wei B;Zhang S;Liu M
慢性淋巴细胞性炎症伴脑桥血管周围增强类固醇反应（CLIPPERS）是一种病因不明的炎症性疾病。副肿瘤的病因可能是一个原因。我们报告一例CLIPPERS与腮腺癌。一位54岁男性，有淋巴瘤病史，因桥小脑综合征住院。脑MRI显示脑桥和小脑“布满”点状和曲线状强化病灶，支持CLIPPERS的诊断。通过进一步的小脑活检显示白质中主要是CD3+ T细胞，排除淋巴瘤复发。患者在静脉注射甲基强的松龙和大剂量皮质类固醇的脉冲治疗后病情缓解，但当皮质类固醇逐渐减少到较低剂量时，他抱怨步态问题恶化。虽然通过增加糖皮质激素与硫唑嘌呤的剂量，临床症状再次逐渐改善，但患者在随访期间仍有轻微的步态不稳。随访7个月，MRI发现腮腺肿块，活检证实为癌。腮腺癌切除后，残留症状及既往MRI病变消失，无复发。CLIPPERS可能不是一个独特的病理科实体，但重叠的诊断与其他疾病。一些CLIPPERS病例可能是副肿瘤神经综合征（PNS）的一种亚型，因为其机制与抗体介导的脑炎相似。对于CLIPPERS患者，尤其是停止或逐渐减少皮质类固醇治疗后复发的患者，建议进行肿瘤筛查和血清副肿瘤自身抗体检测。在线版本包含补充材料，可在10.1186/s12883-021-02135-6获得。
Chronic lymphocytic inflammation with pontine perivascular enhancement responsive to steroids (CLIPPERS) is an inflammatory disorder with unclear causes. Paraneoplastic etiology may be a cause. We report a case of CLIPPERS with parotid carcinoma. A 54-year-old man with a history of lymphoma was hospitalized with a pontocerebellar syndrome. Brain MRI revealed that the pons and cerebellum were “peppered” with punctate and curvilinear enhancement lesions that supported the diagnosis of CLIPPERS. The relapse of lymphoma was excluded by a further cerebellum biopsy revealing predominantly CD3+ T cells in white matter. The patient was relieved after pulse therapy with intravenous methylprednisolone and a large dose of corticosteroids, but he complained of a worsening gait problem when corticosteroids were tapered to a lower dose. Although the clinical symptoms gradually improved again by increasing the dosage of corticosteroids with Azathioprine, the patient still had a slight unsteady gait during follow-up. At the 7-month follow-up, a parotid mass was detected by MRI and was verified as carcinoma by biopsy. After resection of parotid carcinoma, the residual symptoms and previous MRI lesions disappeared, and no relapse occurred. CLIPPERS may not be a distinct nosologic entity but an overlapping diagnosis with other diseases. Some cases of CLIPPERS might be a subtype of paraneoplastic neurological syndromes (PNS) due to the similar mechanism of antibody-mediated encephalitis. Tumor screening and serum paraneoplastic autoantibody tests are recommended for patients with CLIPPERS, especially for those who relapse when corticosteroids treatment is stopped or tapered. The online version contains supplementary material available at 10.1186/s12883-021-02135-6.
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